Complications After Surgical Correction of Anorectal Malformations

Hong Kong J Radiol. 2020;23:176-84 | https://doi.org/10.12809/hkjr2017047

REVIEW ARTICLE

Complications after Surgical Correction of Anorectal Malformations T Hosokawa1, Y Yamada2, Y Tanami1, Y Sato1, Y Tanaka3, H Kawashima4, E Oguma1 1Department of Radiology, Saitama Children’s Medical Center, Saitama, Japan 2Department of Radiology, Keio University School of Medicine, Tokyo, Japan 3Department of Pediatric Surgery, Nagoya University Graduate School of Medicine, Saitama, Japan 4Department of Surgery, Saitama Children’s Medical Center, Saitama, Japan

ABSTRACT Radiologists are often unfamiliar with anorectal malformations and have limited knowledge of the surgical procedures for their repair. In this article, we provide a comprehensible description of the surgical procedures for radiologists, review previous literature, and summarise the incidence of the complications. Moreover, we detail major postoperative complications consequent to the use of various imaging techniques, including anorectal prolapse, anal stenosis, urethral injury, posterior urethral diverticulum, neurogenic bladder, adhesion of reconstructed vagina, leakage from suture lines, and trocar site hernia. Knowledge of these complications and surgical procedures is important to radiologists for diagnosis and determination of a treatment strategy.

Key Words: Anorectal malformations; Anus, imperforate

中文摘要

肛門直腸畸形矯正術後的併發症 T Hosokawa、Y Yamada、Y Tanami、Y Sato、Y Tanaka、H Kawashima、E Oguma

放射科醫師通常不熟悉肛門直腸畸形，並且對其修復的手術程序認識有限。本文為放射科醫生提供全面的手術方法說明、回顧文獻並總結併發症的發生率。此外，我們詳細介紹由於使用各種成像技術顯示主要術後併發症，包括肛門直腸脫垂、肛門狹窄、尿道損傷、後尿道憩室、神經源性膀胱、重建陰道粘連、縫合線滲漏以及套管針疝。這些併發症和手術程序的知識對於放射科醫生診斷和確定治療策略很重要。

Correspondence: Dr Takahiro Hosokawa, Department of Radiology, Saitama Children’s Medical Center, Saitama, Japan Email: [email protected]

Submitted: 5 Nov 2018; Accepted: 3 Dec 2018

Contributors: TH, YY and YTanami contributed to the design of the study. YTanami, YS and YTanaka acquired the data. TH, YY, YTanami, YS and EO performed analysis or interpretation of data. TH and YY wrote the article. HK and EO carried out critical revision for important intellectual content. All authors had full access to the data, contributed to the study, approved the final version for publication, and take responsibility for its accuracy and integrity.

Conflicts of Interest: All authors have disclosed no conflicts of interest.

Funding/Support: This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Ethics Approval: This study is in accordance with the tenets of the Declaration of Helsinki and was approved by the ethics committee of our institution. Informed consent was waived.

INTRODUCTION stenosis, urethral injury, posterior urethral diverticulum, Congenital anorectal malformations (ARMs), also adhesion of reconstructed vagina, leakage from suture known as imperforate anus, affect approximately 1 in lines, neurogenic bladder, and trocar site hernia. 5000 newborns.1 These ARMs are classified as low, intermediate, or high types,1 with treatment based on SURGICAL PROCEDURES this classification.2 Although a variety of treatments Several surgical procedures are performed to repair are available for imperforate anus, almost all cases of ARMs. Innovative approaches such as PSARP by Peña low-type imperforate anus are managed with a one-step and Devries6 and LAARP by Georgeson et al4 have been anoplasty immediately after birth.2,3 In contrast, although reported. The anterior or posterior perineal approach is primary anorectal repair without a diverting enterostomy selected according to fistula location and ARM type. is performed in some patients with intermediate- or The anterior perineal approach is usually selected in low- high-type imperforate anus,3-5 almost all patients with type or anovestibular ARM, and the posterior perineal these types are treated first with a diverting colostomy, approach is usually selected for intermediate-type ARM then anorectoplasty.3-5 Patients with ARMs are treated (Figure 1). with anorectoplasty for complete repair of the ARMs, regardless of type. There are several other approaches INCIDENCE AND DESCRIPTION OF similar to anorectoplasty for complete surgical repair of COMPLICATIONS ARM.4,6-8 Currently, many surgical procedures, such as There are several approaches for surgical repair of ARMs, perineal anorectoplasty, sacroperineal anorectoplasty, and there are numerous reports on related complications. abdominosacroperineal anorectoplasty, posterior We reviewed previous reports on complications after sagittal anorectoplasty (PSARP),6 anterior sagittal surgery for ARM by the abdominal pull-through anorectoplasty (ASARP),8 and laparoscopically assisted approach (Table 117,27), PSARP (Table 29,12,13,15,25-27,31-33), anorectoplasty (LAARP)4 are performed for complete anterior sagittal anorectoplasty (Table 320,23), and LAARP surgical repair of ARM. Despite advances in surgical (Table 410,13,24,29,31-33). Previous reports that included procedures, there are possibilities of postoperative multiple surgical approaches are excluded. The reports complications. exhibit differences with respect to patient sex and ARM type. Therefore, the prevalence of each complication Reports on postoperative complications of surgical shows variations. Furthermore, while the incidence of repair of ARMs have documented the involvement the complications decreases with the improvement in of pelvic organs (such as anus, rectum, urethra, and surgical techniques and skills,10,15,25 some complications vagina) as well as cutaneous structures.9-28 Various still occur when the techniques are applied by highly imaging techniques, such as plain radiography, skilled surgeons. colonography, voiding cystourethrography, ultrasonography, computed tomography, and Table 1. Data in previous reports on complications after surgery magnetic resonance imaging (MRI) can be used for for anorectal malformation by the abdominal pull-through diagnosis.9,11,16,18,19,21,29,30 Unlike surgeons, radiologists approach. are often unfamiliar with ARMs and have little Laberge17* Leva27† knowledge about the surgical procedures for their Study period 1976-1982 2002-2009 repair; to date, only one review article related to No. of patients 27 4 radiography has been published.30 Sex Both Male Type High / High / intermediate intermediate The aim of this article was to familiarise radiologists Surgical procedure AP p-t AP p-t with common complications of specific surgical Anal prolapse 40% 25% approaches and ARM types, which would be useful in Anal stenosis 7% - Urethral injury - - diagnosis and in assisting surgeons with the management Posterior urethral diverticulum - - of these complications. In this article, we provide a Complications with vagina - - comprehensible description of the surgical procedures for Leakage from suture lines - - radiologists, review previous literature, and summarise Neurogenic bladder 11% - Abbreviation: AP p-t = abdominoperineal pull-through. the incidence of complications. Moreover, we describe * All patients with bladder problems had severe sacral anomalies. and discuss eight major postoperative complications † Single-stage surgical correction was performed during the specific to ARM, including anorectal prolapse, anal neonatal period.

Hong Kong J Radiol. 2020;23:176-84 177 Complications after Surgical Correction of ARM

(a) (b) (c) (d)

Figure 1. Schematic presentation of the surgical approaches for repair of anorectal malformation. (a) The pull-through can be performed via the abdominal, anterior perineal (anterior sagittal), or sacroperineal (posterior sagittal) approach. Posterior sagittal anorectoplasty or the sacroperineal approach comprises incising the posterior area of the anal site (dotted yellow line) and performing pull-through (yellow curved arrow). Anterior sagittal anorectoplasty involves incising the anterior area of the anal site (dotted blue line) and performing pull-through (blue curved arrow). The abdominal approach involves performing pull-through after abdominal incision (red arrow). Laparoscopy may be used instead of abdominal incision. (b) Low-type anorectal malformation with anovestibular fistula. In female patients with low-type anovestibular fistula, anterior sagittal anorectoplasty is usually selected as the surgical approach. Anterior sagittal anorectoplasty involves incising the anterior area of the anal site (dotted blue line), performing pull-through (blue curved arrow), and separating the anovestibular fistula (green arrow) from the vestibule. During anterior sagittal anorectoplasty, the fistula is identified without rectal incision under direct vision. (c) Intermediate-type anorectal malformation with rectourethral fistula. In male patients with intermediate-type anorectal malformation with rectourethral fistula, the posterior sagittal anorectoplasty or abdominal approach (including laparoscopically assisted anorectoplasty) is usually selected as the surgical approach. Posterior sagittal anorectoplasty or the sacroperineal approach involves incising the posterior area of the anal site (dotted yellow line), performing pull-through (yellow curved arrow), and separating the rectourethral fistula (green arrow) from the rectum. During posterior sagittal anorectoplasty, the rectum is incised from the dorsal side and the fistula is identified in the rectal lumen. The abdominal approach or laparoscopically assisted anorectoplasty involves performing pull-through from the abdominal side (red arrow) and separating the rectovesical fistula from the rectum. During laparoscopically assisted anorectoplasty, the fistula identified without incision of the rectum under direct vision. (d) High-type anorectal malformation with rectovesical fistula. In male patients with high-type anorectal malformation with rectovesical fistula, the abdominal approach (including laparoscopically assisted anorectoplasty) is usually selected as the surgical approach. The abdominal approach or laparoscopically assisted anorectoplasty involves performing pull-through from the abdominal side (red arrow) and separating the rectovesical fistula (green arrow) from the rectum. Surgical repair of anorectal malformation is divided into two steps: anoplasty and the pull-through step. Anoplasty involves creating a new anus at the correct site. This step is performed at the perineum (asterisks). The pull-through step entails moving the distal rectal pouch to the correct new anal site and anastomosing it with the distal anus. During pull-through, the fistula is separated from the anorectal tract. Abbreviations: R = rectum; B = bladder; V = vestibule.

Table 2. Data in previous reports on complications after surgery for anorectal malformations by posterior sagittal anorectoplasty.

Nakayama9 Hong15* Belizon12 Huang26 Julià25 Leva27 England13 De Vos33 Ming32 Koga31 Study period 1982- 1981- 1980- 1988-2008 1994- 2004- 2002- 2005- 2000- 1992- 2000- 1985 2000 2002 2003 2008 2009 2009 2009 2001 2013 No. of patients 23 572 833 85 103 54 53 19 19 19 34 8 Sex Both Male Both Both Both Both Both Male Both Both Male Male Type All All All Low High Mix Mix High / All High / High Intermediate intermediate intermediate Anal prolapse - - 3.8% 7.1% 15.5% 17% 4% - 10.5% 15.8% - - Anal stenosis - - - 3.5% 13.6% - - 5.3% 21.1% 5.3% 5.9% 12.5% Urethral injury - 3.3% ------Posterior 4.3% 0% - - - 1.9% - - - - 21.3% - urethral diverticulum Complications 4.3% - - - - 1.9% ------with vagina Leakage from 17.4% ------suture lines Neurogenic - 0% ------bladder * This study focused on urological complications and divided patients into two groups based on the institution (posterior sagittal anorectoplasty or several surgical procedures, including posterior sagittal anorectoplasty).

178 Hong Kong J Radiol. 2020;23:176-84 T Hosokawa, Y Yamada, Y Tanami, et al

Anal Prolapse in low-type ARM.26,35 It may be accidentally detected on Anorectal prolapse (Figure 2) is defined as anal prolapse an MRI requested to evaluate the levator ani muscle.31,36 >5 mm.12 There have been no radiographic reports on anorectal prolapse, as this complication is clinically Anal Stenosis diagnosed. Anal prolapse has a significantly higher Anal stenosis (Figure 3) may occur with all surgical incidence in patients with a low quality of the levator procedures and ARM types, and it may be caused by ani muscle and in those with vertebral anomalies,12,22 and ischaemia or inadequate dilation of the anus.7 Ischaemic the frequency of this complication is also reported to be necrosis of the pull-through bowel is a technical problem associated with surgical approaches as LAARP.29,31,32,34 caused by a reduction in vascular supply to the border High-type ARM is characterised by poor muscle after colon mobilisation.17 In abdominal radiography quality, which may render anal prolapse an inevitable after surgical repair of ARM, constipation rather than complication, with a higher likelihood of recurrence than poor levator ani muscle function may be observed, but anal stenosis must still be considered.36,37

Table 3. Data in previous reports on complications after surgery Urethral Injury for anorectal malformation (not including laparoscopically assisted Urethral injury (Figure 4) during surgery has been found anorectoplasty). to occur more often in male patients with intermediate- Zamir23 Wang20 or high-type ARM.15,38 To repair a rectourethral fistula, Study period 2007 2008-2012 separation of the urinary tract from the rectum is required. No. of patients 30 26 Therefore, there is a risk of urethral injury while repairing Sex Female Female Type Mix Low such an ARM, which should be avoided by paediatric Surgical procedure Anterior sagittal Anterior sagittal surgeons.16,19 To prevent injury to the urinary tract, an anorectoplasty anorectoplasty augmented-pressure distal colostogram before surgical Anal prolapse 3.3% 3.8% 15,38 Anal stenosis 6.7% 0% repair is recommended. Urethral injury - - Posterior urethral diverticulum - - Posterior Urethral Diverticulum Complications with vagina - - Posterior urethral diverticulum (Figure 5) is more Leakage from suture lines - - Neurogenic bladder - - likely to occur in LAARP than in the other types of surgery.10 This is important because it may result in

Table 4. Data in previous reports about complications after surgery for anorectal malformation by laparoscopically assisted anorectoplasty. Japanese England13 † Podevin24 De Vos33 Jung29 Ming32 Koga31 multicenter study group on male high imperforate anus10 * Study period 2000-2006 2005-2009 2002-2007 2000-2009 2003-2010 2001-2012 2000-2013 No. of patients 45 24 34 20 25 32 12 Sex Male Both Male Both Male Male Male Type High All High High / High / High Intermediate intermediate intermediate Anal prolapse 44% 4.2% 8.8% 5% 52% 9.4% 50% Anal stenosis 9% 33.3% 23.5% 10% 4% 3.1% - Urethral injury - - - 5% - - - Posterior urethral diverticulum 33.3% - - - 4% - 8.3% Complications with vagina ------Leakage from suture lines 11% 4.2% (perineal 2.9% - - - - abscess) Neurogenic bladder - - 2.9% - - - - Trocar-site hernia - - 2.9% 10% - - - * This study compared the incidence of complications between two different surgical procedures and found no significant differences, except in the posterior urethral diverticula. † This study compared the incidence of anal stenosis and prolapse between two different surgical procedures and found no significant difference.

Hong Kong J Radiol. 2020;23:176-84 179 Complications after Surgical Correction of ARM

(a) (b)

Figure 2. Anorectal prolapse. This male patient presented with a rectovesical fistula with high-type anorectal malformation at age 2 years. Laparoscopically assisted anorectoplasty had been performed as the second surgical repair at age 10 months. After the second surgical repair, anorectal prolapse occurred. Therefore, surgical repair (Gant-Miwa method) was performed at age 2 years. (a) T2-weighted sagittal magnetic resonance imaging showing rectal and anal depression from the pelvic floor to the distal side. Rectal prolapse is located at 7 mm (double headed arrow) from the skin around the anus [dashed line]. (b) Axial T2-weighted magnetic resonance imaging in a male infant aged 6 months with rectovesical fistula. It detects asymmetrical puborectalis muscle only on the left side (arrow), which cannot be clearly visualised.

(a) (b)

Figure 3. Anal stenosis. This female patient presented with an anovestibular fistula with a low-type anorectal malformation at age 10 months. After surgical repair via perineal anoplasty, mucosal necrosis was observed. Severe constipation persisted and the patient was diagnosed with anal stenosis. Subsequent dilation was required. (a) Abdominal radiograph showing dilated rectum and colon. The rectum is full of faeces. (b) Colonography using barium revealed anal stenosis (arrow).

dysuria, formation of urinary stones, infection, and about posterior urethral diverticulum, and in some cases, malignancy.10,16,18,19 Meanwhile, some patients with posterior urethral diverticula could not be revealed using posterior urethral diverticula may not exhibit any voiding cystourethrography, being detectable only using symptoms.10,16,18,19 Therefore, it may be accidentally MRI.10,11,16,18 Histopathology of the excised mucosa of detected on an MRI performed to evaluate the levator ani the cyst showed colonic mucosa and confirmed that cyst muscle.31,36 There have been some radiographic reports was indeed an enlarged residual rectourethral fistula.16

180 Hong Kong J Radiol. 2020;23:176-84 T Hosokawa, Y Yamada, Y Tanami, et al

(a) (a)

(b) (b)

Figure 4. Urethral injury. This male patient presented with Figure 5. Posterior urethral diverticulum. This male patient rectourethral fistula with an intermediate-type anorectal presented with an intermediate-type anorectal malformation malformation at age 1 month. Perineal anoplasty had been at age 6 months and underwent laparoscopically assisted performed on the day after birth. During surgical repair, urethral anorectoplasty as the second surgical repair. At age 2 years, injury occurred, and prolonged indwelling urinary catheterisation magnetic resonance imaging was performed for evaluation of a was required. (a) Voiding cystourethrography performed after cystic lesion anterior to the rectum, which was asymptomatic and surgical repair. Leakage of iodine contrast medium from injury site incidentally detected using ultrasonography. Surgical resection (arrows). This finding was not revealed in the preoperative voiding was performed. (a) Axial T2-weighted image showing cystic lesion cystourethrography (not shown). (b) An indwelling urinary catheter with high signal intensity between the rectum and bladder (arrow). was required and a voiding cystourethrography was performed (b) Sagittal fat-suppression T2-weighted image showing the oval after 3 years. Voiding cystourethrogram showing mild urethral lesion posterior to the urethra (arrow). stenosis (arrow).

To prevent posterior urethral diverticula, novel surgical management of patients with ARM.40 approaches and enhanced surgical skills are required.16,19,39 Adhesion of Reconstructed Vagina Neurogenic Bladder Reconstruction of the vagina may be required in girls Although neurogenic bladder (Figure 6) is a with cloacal malformation, which is classified as high- complication of ARM repair,15,21 spinal anomalies type ARM. Reconstruction of the vagina using the commonly accompany ARMs.35 Laberge et al17 reported intestine has been previously reported,41 with some that three patients had prolonged poor bladder emptying patients requiring dilatation.41 Partial adhesion (Figure 7) and that these patients had severe sacral anomalies. of the reconstructed vagina or uterus must be diagnosed However, determining whether the cause of neurogenic early to reduce decline in quality of life of patients bladder is iatrogenic may be difficult. Follow-up with ARMs.41 Furthermore, some patients may require regarding urological complications is important for additional surgical repair.42-45

Hong Kong J Radiol. 2020;23:176-84 181 Complications after Surgical Correction of ARM

(a)

(b)

Figure 6. Neurogenic bladder and adhesion of reconstructed vagina. This female patient presented with a cloacal malformation at age 12 years. She had undergone surgical repair and vaginal reconstruction using the small intestine at age 1 year. After Figure 7. The same female patient as Figure 6. (a) Vaginography surgical repair, she developed a neurogenic bladder requiring performed at age 12 years showing adhesion within the clean intermittent self-catheterisation. Voiding cystourethrogram reconstructed vagina. Vaginal stenosis was observed (arrow). (b) showing the bladder wall with several diverticula and a large Vaginography after balloon dilatation showing that the proximal volume. side of the vagina was dilated.

Leakage from Suture Lines than 90% of trocar site hernias are within 10 mm,46,47 Leakage from suture lines (Figure 8), failed and they have occurred in paediatric patients.24,33 Some anastomoses, and perineal abscesses has been cases may need surgical repair because of small bowel reported.9,10,13,14 Infection after operation is a common obstruction with strangulation caused by a port site complication,9,10,13,14,20 and the diagnosis of leakage hernia.48 If this complication is detected, radiologists from the suture line is important in determining the should evaluate the possibility of bowel strangulation. best treatment option. Leaks may be detected with To prevent this complication, laparoscopic port closure colonography,9 whereas only one report has included is usually performed using different techniques.49,50 For radiographic images.9 Fistula repair can be achieved via radiologists, knowledge of trocar site hernia is important colonostomy, antibiotic therapy, and spontaneous self- for early diagnosis. closure.9,13,14 CONCLUSION Trocar Site Hernia We have described eight complications after surgery for LAARP is associated with trocar site hernia (Figure 9). ARM. These complications involve the pelvic organs. Previous studies have shown that the incidence of this Various imaging techniques are used to diagnose these complication ranges between 1% and 10%.24,33,46,47 More complications. Although the incidence of these types

182 Hong Kong J Radiol. 2020;23:176-84 T Hosokawa, Y Yamada, Y Tanami, et al

(a) (b)

Figure 8. Leakage from suture line. This female patient presented with anovestibular fistula with a low-type anorectal malformation at age 1 year. She was discharged from the hospital after undergoing perineal anoplasty. Then, her buttocks began to swell because of an abscess. Colonography showing the presence of a fistula extending from the suture line to the skin. She underwent colostomy and antibiotic therapy was initiated. (a) Lateral radiograph with colonography using barium showing anorectal cutaneous fistula extending from the suture line (arrow). (b) Sagittal reformatted contrast computed tomography showing a low attenuation area surrounding an enhanced rim posterior to the rectum (arrow). In this lesion, gas was detected, and an abscess was identified.

(a) of complications varies across reports, knowledge of their manifestation and treatment is important for radiologists.

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